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Health outcomes for children with neurodisability: what do professionals regard as primary targets?
  1. Astrid Janssens1,
  2. Jane Williams2,
  3. Richard Tomlinson3,
  4. Stuart Logan1,
  5. Christopher Morris1
  1. 1Peninsula Cerebra Research Unit (PenCRU) & NIHR PenCLAHRC, University of Exeter Medical School, Exeter, UK
  2. 2Nottingham University Hospitals NHS Trust, Nottingham, UK
  3. 3Royal Devon & Exeter NHS Foundation Trust, Exeter, UK
  1. Correspondence to Dr Christopher Morris, PenCRU, Child Health Group, University of Exeter Medical School, Veysey Building, Salmon Pool Lane, EX2 4SG, Exeter, UK; christopher.morris{at}exeter.ac.uk

Abstract

Aim To identify what aspects of health clinicians target when working with children with neurodisability, and which might be appropriate to assess the performance of health services.

Method Health professionals were recruited through child development teams and professional societies in England. Professionals participated in four rounds of an online Delphi survey. Open questions were used to elicit aspects of health; these were coded using the WHO International Classification of Functioning, Disability and Health for Children and Youth. Then, participants were asked to rate their agreement with statements to prioritise outcomes identified.

Results Responses to all four rounds were, respectively: 233/276 (84.4%), 232/286 (81.1%), 227/285 (79.6%) and 191/284 (67.3%). The key outcome domains identified were: mental health, confidence/emotional stability, anxiety/attention, sleep, pain, toileting, movement ability, manual ability, acquiring skills, communication, mobility, self-care, recreation and leisure. Participants rated both functioning and well-being in these aspects of health as equally important.

Interpretation This Delphi survey identified nine key domains that provide a professional perspective on a core set of outcomes for evaluating services for children and young people with neurodisability.

  • Health services research
  • Outcomes research
  • Neurodisability
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What is already known on this topic

  • Providing high-quality multidisciplinary services requires professionals to work collaboratively towards shared objectives.

  • A common vision among professionals for what health services seek to achieve for children has been noted as a barrier to improving health services.

  • Outcomes are increasingly used as performance indicators to examine and compare effectiveness and efficiency of healthcare organisations.

What this paper adds

  • Professionals working with children with neurodisability reached consensus on a core set of aspects of health that are primary concerns for health services.

  • Professionals perceive themselves more as being ‘accountable’ primarily for delivering against the more clinical rather than social participation and well-being domains.

  • Professionals’ primary targets show partial overlap with what families regard as important, including pain, communication, mobility, self-care and emotional well-being.

Introduction

Estimates of the prevalence of childhood disability in the UK vary from 5% to 18% depending on the definition or indicator used.1 Commonly, an estimate of one in 20 children is cited.2 Neurodisability is an umbrella term for conditions associated with impairment of the nervous system and includes conditions such as cerebral palsy, autism and epilepsy.3 Aside from asthma, neurodisability is thought to represent the largest proportion of significant childhood disability.4

Although neurodisability represents a heterogeneous group of conditions, they share much in common in terms of healthcare needs. Children and young people affected by neurodisability have a range of impairments some of which are relatively minor, but many have complex healthcare needs. As a consequence, many are frequent and intensive users of the health system, and have contact with an average of at least ten different health professionals.5 ,6 Providing high-quality multidisciplinary services requires professionals to work collaboratively towards shared objectives.7 However, merely acknowledging and agreeing to work in partnership is not sufficient: a common vision is needed for an efficient integrated health service, strong enough to bind all providers and services.7 One key barrier to improving health services for children and young people is that health professionals often hold differing views of their responsibilities.8

Performance indicators are increasingly mandated to examine and compare effectiveness and efficiency of healthcare organisations. Proposed indicators include routinely collected clinical data and patient reported outcome measures (PROMs).9 Hence, agreement on the aspects of health that clinicians are seeking to change would provide a professional's perspective on a core suite of outcomes to assess the performance of health services.

The purpose of this study was to identify which aspects of health clinicians target when working with children with neurodisability, and which aspects of health they regard as the primary aims of health services. This was part of a programme of research commissioned by the UK Department of Health to inform the National Health Service (NHS) Outcomes Framework, which also included a systematic review of PROMs for children and young people, and qualitative research with young people and parents.10

Methods

Recruitment and sampling

The multidisciplinary sample of health professionals was recruited initially through an invitation sent to Child Development Teams in England. A subsequent phase of purposive sampling sought to recruit under-represented professions; several professional societies forwarded invitations to their members (eg, for paediatric neurologists, surgeons, ophthalmologists, physiotherapists, speech and language therapists, and mental health professionals). Participants registered to take part using an online form; they provided their profession, region of England where employed and email address.

The Delphi process

Data were collected using four iterative rounds of an online Delphi survey, an established method for seeking consensus.11 Recommended procedures were followed, including beginning with open questions before moving to more specific closed questions, and retaining non-responders in subsequent rounds.12 ,13 Most participants were identified at the start of the process, but a small number of professionals approached researchers between Rounds 1 and 2 were included in subsequent rounds. In each round, participants were emailed a unique link to an online survey system. Participants were presented with a mix of open questions with free text response boxes, or asked to rate their agreement with statements using a 4-point Likert scale: ‘strongly disagree’, ‘disagree’, ‘agree’ and ‘strongly agree’. Non-responders were sent up to two reminder emails, which included an option to withdraw.

Survey questions

In Round 1, participants were invited to state, using free text, what aspects of health they tried to influence and/or improve when working with children and young people with neurodisability; examples (‘improve communication’, ‘reduce pain’, etc) were provided in order to encourage respondents to be specific. Free text responses were coded with reference to the WHO International Classification of Functioning, Disability and Health for Children and Youth (ICF-CY).14

In Round 2, participants were presented with the list of ICF-CY components of health identified in the first survey. Participants were invited to rate their agreement with the list as broadly covering health outcomes that the NHS should routinely assess for children with neurodisability. They were also invited to suggest any additional aspects of health that they felt should be considered.

In Round 3, participants were presented with a revised list. Participants were again invited to rate their agreement with the list as appropriate for measuring NHS outcomes. We also asked whether they sought to improve ‘functioning’ (as in what people can do) and/or ‘well-being’ (as in how people feel about their health).

In Round 4, we sought views on aspects of health specifically assessed by PROMs for children and young people identified in a systematic review.15 In that study, domain scales and items were also coded using the ICF-CY,14 identifying 23 discrete health domains. Respondents were asked to rate to what extent they thought the health service should be responsible for these outcomes on a seven point scale (from ‘not at all’ to ‘completely’).

Analysis

Responses in each round were categorised and discussed by the team to inform further rounds of the survey. We used an a priori criterion of 67% approval (agree or strongly agree) to indicate broad consensus among participants.13

Results

Survey response

In total, 295 health professionals from a range of professions registered. Eight participants withdrew from the study in the first round, and one opted out at each further round. Responses were: 233/276 (84.4%), 232/286 (81.1%), 227/285 (79.6%) and 191/284 (67.3%) (figure 1 and table 1).

Table 1

Profession of respondents to each individual round

Figure 1

Flowchart showing responses to the Delphi survey.

Identifying aspects of health

In Round 1, 233 respondents suggested 1524 aspects of health they sought to influence. Some respondents stated general terms, such as, ‘improve function’, ‘maintain/improve comfort’, ‘maintain/increase participation’, ‘improve quality of life’ and ‘help the child have fun’. Others were more specific: ‘increasing fine motor skills’, ‘improving motor coordination’ and ‘improving gross motor skills: rolling, sitting, moving on the floor, crawling, standing, walking, running, jumping, coordination, balance, core stability etc.’

All 1524 aspects of health were mapped to the ICF-CY.14 As a group, respondents reported targeting components across the ICF. In addition, some respondents reported processes, such as ‘making referrals’. Some aspects of health were cited more frequently; aspects of health that were mentioned 20 times or more are presented in table 2.

Table 2

Aspects of health mentioned 20 times or more by healthcare professionals in Round 1, ordered by frequency mentioned (list as presented in Round 2)

When, in Round 2, participants were presented with the list of most commonly cited aspects of health (table 2), 211/232 (90.9%) of respondents agreed or strongly agreed that the list broadly included at least some of the aspects of health they target. In addition, 207/232 (89.2%) agreed or strongly agreed that these were appropriate to use as health outcomes; nevertheless, we received useful recommendations to refine the list.

In Round 3, 209/227 (92%) respondents agreed or strongly agreed with the revised list (see box 1). Feedback in Round 3 concerned largely presentation issues, for example, adding or changing the examples. In terms of focus for professionals, 212/227 (93.4%) indicated they targeted ‘functioning’ in relevant domains and 190/227 (83.7%) indicated they targeted ‘well-being’.

Box 1 List of aspects of health as presented in Round 3

  • Mental functions: for example, mental health issues

  • Consciousness functions: for example, seizures, stroke

  • Temperament and personality functions: confidence, emotional stability

  • Sleep

  • Specific mental functions: for example, anxiety, attention

  • Psychomotor control: for example, behavioural problems

  • Pain

  • Functions related to digestive system: for example, constipation, swallowing, drooling

  • Urinary functions (continence, enuresis)

  • Neuromusculoskeletal and movement: gross and fine motor function; quality of movement

  • Mobility of joint functions: for example, mobility and ease of movement of joints

  • Muscle power functions: muscle strength

  • Muscle tone function: for example, spasticity

  • Control of voluntary movement functions: for example, movements, head and trunk control

  • Learning and applying knowledge: acquiring skills; learning to read, write

  • Acquiring basic skills: for example, fine motor function to improve eating, pencil holding

  • Communication

  • Changing and maintaining body position: for example, sitting, standing, lying down

  • Mobility (in activity and participation): for example, moving, making transfers between posture

  • Self-care: for example, independence in all activities of daily living (washing, toileting, dressing, eating)

  • Community, social and civil life: for example, engaging in social clubs; recreation and leisure

  • Movement (body structures and functions): for example, gait, deformity; muscle length and joint range of movement

Rating of health targets

In Round 4, respondents rated the following domains as being the primary responsibility of health services: pain, hearing, seeing, vision, mobility, communication, manual ability, sleep and toileting. Those aspects of health that were rated as less of the responsibility of health services were: play, relationships with family, sport and leisure, learning and applying knowledge, and relationships with friends (table 3). No aspects of health were rated as ‘not the responsibility of health services’.

Table 3

Professionals’ rating of NHS responsibility for aspects of health assessed by PROMs

Discussion

The Delphi survey provides an insight into what current health professionals target as health outcomes for children with neurodisability. The participants who were a multidisciplinary group of health professionals mentioned targeting items across ICF domains. Professionals generally agreed that these ICF components should be assessed routinely. Health professionals rated as the primary responsibility of the health services: pain, hearing, seeing and vision, mobility, communication, sleep, emotional well-being, toileting, self-care, and sexual health. Similar aspects of health were identified by adolescents with cerebral palsy and parents as important for ‘quality of life’.16

Less endorsed by consensus as a primary concern for health services were: play, relationships with family and friends, sport and leisure, and learning and applying knowledge. When asked whether the healthcare system should target well-being (how a patient feels about his or her health) or functioning (what a patient can do), the majority of participants indicated that both were important.

In the early phases of the process, health professionals in our study identified aspects of social and emotional functioning as important in addition to more ‘clinical’ domains. However, when asked to rank which domains were more the responsibility of health services, they prioritised those related to the ICF domains body functions and structures.

Participants in this study appeared to recognise the importance of a broad range of aspects of health for children, but the findings suggest that professionals perceive themselves ‘accountable’ more for medical issues rather than social participation and well-being. This may help explain apparent discordance between the goals of professionals and families, despite wide recognition that the overall objective is quality of life.17–19 In other words, the discrepancies in focus between professionals and families may reflect not so much differences in overall objectives, but rather what professionals see as their role. It may also reflect anxieties about measurement of these outcomes, given the extent to which they believe their current efforts affect these different aspects of health.

In parallel, we explored the views and opinions of children and young people with neurodisability and parents.15 The findings showed partial overlap, as families also highlighted communication, mobility, pain, self-care and emotional well-being.20 However, families highlighted other key domains that were less emphasised by professionals, such as temperament (anxiety and behaviour), interpersonal relationships, community and social life, and gaining independence/future aspirations.

Our findings suggest that health professionals involved in the care for children with neurodisability agree generally on several health domains as primary concerns, and these can be used as health service outcomes. Some of these aspects of health can perhaps be measured using PROMs: mobility, sleep, pain, self-care, movement ability, manual ability, mental health, confidence/emotional stability, anxiety/attention, acquiring skills, communication and social well-being; other aspects might be better clinically assessed.

Many participants recognised limitations to the extent to which they felt health services could be responsible for aspects of health that are influenced by multiple factors. Participants noted the need to discuss treatment and therapeutic goals with families, and negotiate priorities. Thereby, perhaps, professionals are seeking to have at least some influence on those aspects of health more distal to health services’ sphere of direct influence; that is, in contrast to managing pain, which is more clearly perceived as the direct responsibility of health services.

The ICF has encouraged acceptance of a broader conceptualisation of health. However, when attempting to assess the effectiveness of health services it may be appropriate to focus on narrower, more proximal goals. This does not obviate the need to take a broader perspective when planning how integrated services should work together as a system to improve children's health and well-being.

Participants included paediatricians, paediatric neurologists and surgeons, nurses, speech and language therapists, psychologists, and a wide range of other allied health professionals working in different regions of the country participated. This multidisciplinary and geographically diverse (in England) sample is notable. This sample, with 20 different professions represented in each of the four rounds, reflects the variety of health professionals with whom these children interact.

Nevertheless, their views may or may not accurately represent the views of health professionals more broadly. We recognise that our participants were likely to be those motivated to volunteer and take part in the study, which may mark them as different to their colleagues. However, it has been suggested that people who are willing to participate in expert panels do tend to be representative of their colleagues.21 Low response rates can impact the generalisability of a survey's findings. After four rounds, this study still achieved a reasonable response of 67%.22 Although responses dropped in the fourth round, the diversity of professions was retained.

The profession of respondents may have influenced the frequency with which aspects of health were mentioned in the survey. There is a risk that the skewed balance of professions, composed of mainly community and hospital-based professionals, may have biased the results. Professionals working in children's mental health services were less well represented. We did not seek to recruit GPs (family physicians). In addition, the online form for healthcare professionals to register for the study included a limited list to describe their profession. Although there was the option to specify under ‘other’, this might not have identified certain specialisms.

We used WHO ICF-CY terminology and definitions to code the information on aspects of health, and then presented these ICF terms back to participants without definition. While no participants raised this as an issue, and there is increasing awareness of the ICF and related terminology, it is possible that professionals may have interpreted aspects of health terms inconsistently.

Our multidisciplinary sample of health professionals agreed broadly on a core set of domains that can represent appropriate outcomes for health services. These aspects of health overlap to some extent with families’ priorities. The findings offer professionals and services a chance to reflect and benchmark their current practice and therapeutic focus against the broad consensus of their peers. The list, including key aspects of health across the ICF, could be used to help multi-disciplinary teams consider what resources, skills and expertise are needed to provide a holistic approach.23

Sir Ian Kennedy recommended that a common vision be developed for what the NHS is trying to achieve for children and young people.8 The findings in this study provide an incremental step towards suggesting a vision among health professionals for what they target more for children and/or young people with neurodisability. Further efforts are required both nationally and locally to ensure teams work cooperatively and efficiently to achieve agreed outcomes. It is also crucial that families’ views are integrated into the 'shared vision’, both generally and when setting goals for individual children and young people, consistent with the principles of family centred services.24–26 This is likely to lead to greater satisfaction with services, which Kennedy recommended as the ultimate quality indicator.8

Families value highly emotional well-being and social participation, while these were less prioritised by professionals. Although many participants stated they targeted these aspects of health, they felt health services are less accountable for them. This reflects the lack of parity that the English mental health strategy ‘No Health without Mental Health’ sought to change. Healthcare for people with long-term conditions could be improved by better integrating mental health support with primary care and chronic disease management.27 This would appear to require a cultural shift in focus for health professionals and systems alongside clear policy directives. Further research is needed on how best to measure aspects of health agreed by professionals and families as important.

Acknowledgments

This study was part of research funded by the National Institute for Health Research (NIHR) Health Services and Delivery Research programme (Project 10/2002/16); the work also benefited from support from NIHR Collaboration for Leadership in Applied Health Research and Care of the South West Peninsula (PenCLAHRC), and the charity Cerebra. The views and opinions expressed in this paper are those of the authors and not necessarily those of the NHS, the NIHR, the Department of Health or Cerebra. We are very grateful to the participants who gave their time to complete the different rounds of the survey, to the Child Developments Team leaders and professional organisations for forwarding our invitation to participate.

References

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Footnotes

  • Contributors All authors contributed to the conception and design, data collection, interpretation and preparation of the manuscript. All authors have approved of the final version submitted for publication.

  • Funding This study was part of research funded by the National Institute for Health Research (NIHR) Health Services and Delivery Research programme (Project 10/2002/16); the work also benefited from support from NIHR Collaboration for Leadership in Applied Health Research and Care of the South West Peninsula (PenCLAHRC), and the charity Cerebra. The views and opinions expressed in this paper are those of the authors and not necessarily those of the NHS, the NIHR, the Department of Health, or Cerebra.

  • Competing interests All authors have completed the ICMJE uniform disclosure form online (http://www.icmje.org/coi_disclosure.pdf).

  • Ethics approval The UK National Research Ethics Committee North East–County Durham & Tees Valley approved the procedures for the study (reference 11/NE/0364).

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Data sharing statement Further data is available in the full report available through the NIHR Library, or by contacting the corresponding author.

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